Vallecular cyst as a cause of congenital stridor: report of five patients

Background  Vallecular cysts are an unusual cause of congenital stridor. Objective  To describe the imaging findings in five patients, with emphasis on the usefulness of sonographic studies. Materials and methods  Between 1990 and 2007, five patients with a cystic lesion situated in the anterior neck, at the vallecular space, were seen in our institution. Clinical records and imaging findings were retrospectively reviewed. Results  All patients presented with persistent inspiratory stridor that was present from the first week of life. Neck US was performed as part of the investigations in four and showed a vallecular cyst. The diagnosis was confirmed with flexible bronchoscopy in four infants and CT in one; all were resected. Pathology showed a multilayered epithelial border with normal thickness and differentiation; there were no signs of malignancy. Conclusion  Although vallecular cysts are very rare, they should be considered in the differential diagnosis of congenital stridor. When the commonest causes have been ruled out, neck US may be diagnostic. The diagnosis can be confirmed with flexible bronchoscopy or further imaging such as CT or MRI.     

Thyroglossal duct cyst accompanied by laryngomalacia and pulmonary sequestration

A 2‐month‐old full‐term female infant developed nasal stridor, which progressed to respiratory distress and poor sucking ability. Direct pharyngoscopy showed laryngomalacia and a midline cystic mass in the lingual region. The mass pressed on the epiglottis, causing dyspnea. Computed tomography incidentally revealed extralobar pulmonary sequestration. Direct deroofing of the lingual cyst and plication of the epiglottis were performed at 3 months of age, and the patient recovered from the respiratory distress. Histopathology of the cystic mass showed a thyroglossal duct cyst. Thoracoscopic resection of the pulmonary sequestration was then done at 17 months of age. Thyroglossal duct cysts in the lingual region may cause destabilization of the epiglottis and laryngomalacia, resulting in acquired respiratory obstruction. The combination of thyroglossal duct cyst, laryngomalacia, and pulmonary sequestration is rare; therefore, reports must be accumulated in order to explore the embryological origins of such cases.     

23例2个月内小儿舌甲状舌管囊肿的临床特征

目的 通过分析2个月内小儿舌甲状舌管囊肿（lingual thyroglossal duct cysts,LTGDC）的临床特点,提高诊治水平.方法 对23例2个月内的小儿LTGDC与对照组20例1～7岁小儿颈部甲状舌管囊肿（thyroglossal duct cysts,TGDC）的临床资料进行对比分析.结果 23例2个月内小儿LTGDC的临床表现与对照组TGDC患儿明显不同,LTGDC以喉喘鸣为主要表现,有74％以呼吸困难急诊就诊,易误诊为先天性喉软化症或喉软化症合并肺炎;病变位于舌骨后上舌根后方,因突向喉咽腔易导致喉阻塞;电子喉镜检查为首选,有助于鉴别喉软化症、会厌囊肿等;喉部CT/MRI检查为边界清楚的类圆形低密度影,对于确诊未完全突出至喉咽腔的、喉镜下容易漏诊的LTGDC非常重要;治疗采用经口支撑喉镜下手术,手术微创,颈部无瘢痕.20例TGDC均以颈前中线包块为首发症状;颈部超声检查为首选,病变在超声下为边界清楚的液性暗区;手术需采取颈部经皮切开手术.结论 2个月内小儿LTGDC临床表现不同于TGDC,是喉喘鸣伴呼吸困难的常见原因,容易误诊为先天性喉软化症,救治不及时易窒息死亡,应该引起足够的重视.     

新生儿舌甲状舌管囊肿的早期诊断10例报告

目的 使新生儿医生认识和重视新生儿舌甲状舌管囊肿(LTDC),从而避免将LTDC误诊为先天性单纯性喉喘鸣(即喉软化症).方法 总结2004年12月至2007年8月,我院新生儿科收治的10例LTDC患儿,本组10例在院外均误诊为先天性喉喘鸣.入院后5例进行了喉镜检查,10例全部进行了喉部CT扫描,7例进行组织病理学检查.结果 10例均证实为LTDC.喉喘鸣为新生儿LTDC的首发症状,吸气性的呼吸困难是其主要表现;喉镜检查:可见舌根部圆形囊性肿物,色略灰白,表面光滑柔软;喉部CT扫描显示:舌根部圆形低密度灶,直径约0.8～2 cm,边界清楚,凸入气道;组织学检查:囊肿壁南鳞状上皮和柱状上皮组成.结论 LTDC足新生儿甲状舌管囊肿的常见形式,喘鸣及呼吸困难是其主要表现;喉镜检查可作为新生儿LTDC的初筛手段,而喉部CT扫描则提供重要的诊断依据;囊肿穿刺虽可缓解症状,手术切除足LTDC的根治方法 .     

Neonatal stridor and laryngeal cyst: Which comes first?

Neonatal stridor is a rare condition usually caused by laryngomalacia. Congenital laryngeal cyst represents an uncommon cause of stridor in the neonatal population and may be misinterpreted as laryngomalacia, leading to serious morbidity and mortality if diagnosis and treatment are delayed. Herein we report the case of a full‐term infant with stridor, feeding problems and failure to thrive. Initially, direct laryngoscopy diagnosed only laryngomalacia. As stridor worsened, however, and respiratory distress appeared, repeat laryngoscopy showed vallecular laryngeal cyst, visible macroscopically. The patient was successfully treated with endoscopic marsupialization. There was no evidence of recurrence at follow up after 3 months. This case highlights the importance of laryngoscopic assessment for suspected laryngeal abnormalities in infants with stridor. If symptoms worsen, endoscopy should be repeated, because congenital laryngeal cysts may not be immediately visible macroscopically.     

Vallecular cyst: an uncommon cause of stridor in newborn infants

Vallecular cyst, a rare but generally benign lesion in the larynx, may cause stridor and even life-threatening airway obstruction in early infancy. We retrospectively studied 14 cases of newborn infants with vallecular cyst. There was no gender predilection and most cases were full-term and appropriate for gestational age. The clinical presentations included stridor, chest wall retraction, feeding difficulties and failure to thrive. Laryngomalacia was the most common associated anomaly. Flexible laryngoscopy was sufficient for diagnosing the vallecular cyst and larygmalacia. Maintenance of airway patency, nutritional support, and de-roofing of the cyst were the mainstays of management. Conclusion Vallecular cyst should be included in the differential diagnosis of stridor in newborn infants. Respiratory and feeding difficulties in these patients can be dramatically improved after appropriate surgical removal of the cyst. Received: 20 February 1999 / Accepted: 25 June 1999     

婴儿前囟门皮样囊肿

目的 探讨少见的婴儿前囟门皮样囊肿的诊断和治疗。方法 总结6年中经手术后病理证实的婴儿前囟门皮样囊肿12例，男7例，女5例，年龄2个月-2岁，小于l岁9例。临床表现为生后即存在的前囟门肿块，逐渐增大。MRI T1加权呈低信号，T2加权呈高信号。结果 全部病例均行手术摘除，采用弧形切口，保留皮肤，完整剥离囊肿，随访无一例复发。结论 前囟门皮样囊肿是良性先天性发育异常疾病，诊断明确后手术治疗效果好。诊断需与颅顶部的脑膜膨出、颅骨静脉窦、表皮样囊肿相鉴别。     

Saccular cyst in an infant: an unusual cause of life-threatening stridor and its surgical treatment

Saccular cysts in infants are rare lesions of the larynx and may result in respiratory obstruction and severe dyspnea. Herein we present a case of saccular cyst in a three-month-old infant. She was presented with severe stridor and respiratory distress. She had been followed with the diagnosis of tracheomalacia for three months elsewhere. Direct laryngoscopy of the larynx revealed a saccular cyst. Stridor disappeared shortly after surgical excision of the supraglottic saccular cyst under direct laryngoscopy. No complication or recurrence was seen in the 12-month follow-up period. Saccular cysts in infants are rare lesions and should be kept in mind in the differential diagnosis of stridor in infants.     

小儿肠系膜乳糜囊肿

目的探讨小儿肠系膜乳糜囊肿的临床特点及诊治方法。方法回顾性分析我院1987～2003年经手术治疗及病理检查证实的6例小儿肠系膜乳糜囊肿的临床表现及治疗经过。结果本组男4例，女2例，年龄7个月～10岁（平均3岁10个月）。病程30h～2年，病程在7d以内4例。5例表现为急腹症，1例表现为慢性复发性腹痛。术中证实空肠系膜囊肿4例，回肠系膜囊肿2例，囊肿压迫肠管并发肠梗阻2例，并发肠扭转1例，并发乳糜腹2例。3例行囊肿及部分肠管切除吻合术，3例行单纯囊肿切除术，囊内液均呈乳糜状，苏丹Ⅲ染色及乳糜试验阳性。结论小儿肠系膜乳糜囊肿主要表现为腹痛伴腹腔囊性包块，常以急腹症为首发表现，手术切除囊肿及受累肠管预后良好。     

Diagnosis and treatment of lingual thyroglossal duct cyst in newborns

Background:  The objective of this study was to explore the diagnosis and treatment method of lingual thyroglossal duct cyst in newborns.
Method:  The clinical data of nine newborns who were diagnosed as lingual thyroglossal duct cyst were retrospectively analyzed.
Results:  One lingual thyroglossal duct cyst was found when the tongue was pressed with a spatula. The other eight lingual thyroglossal duct cysts were found with a laryngoscope. Three-dimensional computed tomography showed that the cysts were located at the base of tongue, which was round and smooth. Six of nine patients were treated with the puncture method. The fluid was drawn out, and the average volume was 1.4 mL. Follow-up survey lasted for 1 year or more and 33.3% (2/6) of the cases recurred. For the recurrence, the two patients underwent another operation in which most of the cyst walls were removed and they had no recurrence after another year of follow-up survey. Three of the nine patients were treated with the excision method, and they had no recurrence after 1 year of follow-up survey.
Conclusions:  The laryngoscope and three-dimensional computed tomography examinations are important for the diagnosis of lingual thyroglossal duct cyst. Surgical removal serves as a radical cure for lingual thyroglossal duct cyst.     

Mesothelial cyst of the liver in a neonate

Mesothelial cysts are very rare congenital cystic lesions that are derived from coelomic remnants. We report a neonate with a mesothelial cyst of hepatic origin. On routine antenatal ultrasound study, an intra-abdominal mass was seen. After birth, the cystic lesion’s size increased to 8 cm and became symptomatic. Surgical resection was performed. Preoperative diagnostic imaging studies, including CT and MRI, did not identify the etiology, but the cyst appeared to be of hepatic origin. Intra-operatively, the cystic lesion was confirmed to have originated from the liver. Based on immunohistological analysis, the final diagnosis was mesothelial cyst of the liver.     

Congenital pancreatic cyst arising from occlusion of the pancreatic duct

Congenital pancreatic cysts, particularly solitary cysts, are rare causes of abdominal masses in newborns. The etiology of these cysts is unknown and they can vary in location within the pancreas. We present a novel case of an infant with a solitary congenital cyst of the pancreatic duct, itself, as a result of occlusion of the pancreatic duct. Solitary congenital pancreatic cysts are a rare cause of abdominal masses in infants. We recently evaluated a 7-week-old infant with progressive abdominal distension since birth. This child was found to have a large solitary pancreatic cyst. This cyst resulted from occlusion of the main pancreatic duct at the level of the ampulla resulting in cystic degeneration of the pancreatic duct. Based on our review of the literature we believe this is the first documented case of such a cyst.     

Percutaneous drainage as the treatment of choice for neonatal ovarian cysts

Background Involution of neonatal ovarian cysts occurs usually by 12 months. Persisting cysts larger than 4 cm are prone to torsion. Two modes of therapy are advocated: surgery and percutaneous US-guided cyst aspiration.Objective To compare ovarian preservation following the use of US-guided aspiration or conventional surgery for the treatment of large asymptomatic neonatal ovarian cysts, and to suggest alternative treatment when intrauterine ovarian torsion occurs.Materials and methods The study population comprised 25 baby girls with an ovarian cyst, 5 with a simple cyst and 20 with a complex cyst. Of these 25 infants, 8 had surgery and 17 had US-guided cyst aspiration.Results In the surgical group of 8, 6 underwent oophorectomy, and in 2 the ovary was saved. In the aspirated group of 17, the ovary was saved in 10, and was lost in 5. At the time of this report one patient was still in the follow-up period, and one was lost to follow-up.Conclusion US-guided aspiration of large neonatal cysts preserves ovarian tissue in a higher percentage of patients than surgery. It is safe, effective, and repeatable. We recommend US-guided aspiration of asymptomatic large ovarian cysts for salvage or for decompression if intrauterine ovarian torsion occurs. Surgery should be reserved for patients with acute torsion, intestinal obstruction and intestinal volvulus.     

Stridor and gastroesophageal reflux in infants

A relation was found between persistent stridor and gastroesophageal reflux in seven infants, aged 6 weeks to 6 months. Stridor began at 11 days to 2 months of age, and four of the seven infants had transient hypercarbia on at least one occasion before study. Only one had a history of frequent vomiting; three had recurrent pneumonia. Midesophageal pH, chest and abdominal movement, exhaled carbon dioxide partial pressure, and heart rate of six of the infants were recorded for 4 to 12 hours as they slept. Esophageal pH of the seventh infant was recorded for 24 hours. In the six completely studied infants, there were persistent increases of greater than 10 mm Hg in exhaled carbon dioxide level (three infants), of greater than 10 breaths per minute in respiratory rate (four infants), and in retractions and stridor (six infants) 5 to 20 minutes after onset of reflux. Stridor improved with medical management in 48 hours (five of five infants) and disappeared in 3 weeks (three of five infants) to 2 months (one of five infants). One of these medically treated infants subsequently was treated by Nissen gastric fundoplication because of a recurrence of persistent and severe stridor. Three infants had antireflux surgery, and in two of these stridor disappeared in 48 hours. In the third infant stridor disappeared 3 weeks after surgery. Based on this experience, reflux occasionally causes stridor, probably because of acute inflammation of the upper airway. If structural anomalies are ruled out, infants with severe stridor should be examined for gastroesophageal reflux.     

声门下囊肿5例

目的:总结儿童声门下囊肿的临床特征及诊疗经验。方法:对2018年4月至2019年6月山东大学齐鲁儿童医院确诊的5例声门下囊肿患儿的临床资料进行回顾性分析。结果:5例患儿中2例为早产儿,3例为足月儿,均有气管插管史。1例一直无症状,其余4例有喉鸣,其中2例伴声嘶、呼吸困难,曾被误诊为喉软化症、喉炎等。支气管镜检查见声门下...     

Differentiation between pediatric spinal arachnoid and epidermoid-dermoid cysts: is diffusion-weighted MRI useful?

Background Appropriate differentiation between epidermoid-dermoid and arachnoid cysts is important for patient management. MRI has had limitations in differentiating these two types of cysts, especially in the region of the spinal cord. Objective To evaluate the role of diffusion-weighted (DW) MRI for differentiation between spinal arachnoid and epidermoid-dermoid cysts. Materials and methods Four children underwent conventional and DW MRI. Turbo spin-echo imaging was used to obtain DW images. Results On conventional MRI, low T1-W and high T2-W signal was present in all lesions. DW imaging showed restricted diffusion in two patients with epidermoid-dermoid cysts and no water restriction in two patients with arachnoid cysts. Three of four children had extramedullary lesions and one patient had an intramedullary lesion. Three children had surgery, two with epidermoid-dermoid cysts and one with arachnoid cyst. Diagnosis was confirmed by histopathology. One child with a thoracic arachnoid cyst had stable clinical imaging findings during a follow-up of 21 months. Conclusion On conventional MRI, epidermoid-dermoid and arachnoid cysts have similar signal characteristics. Initial experience with spine DW imaging shows promise in differentiating epidermoid-dermoid cysts from arachnoid cysts. Preoperative differentiation is important because it changes management and surgical approach.     

Torted ovarian cyst with lethal bleeding diathesis in an infant

We report a 9-month-old infant with a torted ovarian cyst who presented with an acute consumptive coagulopathy (CC) with lethal outcome. That ischemic tissue can act as a trigger for a CC is well-known, but we did not find any report of a torted ovarian cyst causing a coagulopathy in the pediatric literature. This potential complication constitutes one more reason for the prompt surgical removal of torted ovarian cysts in infants. Accepted: 30 March 1998     

Neonatal neurosonographic correlates of cerebral palsy in preterm infants

Serial neurosonographic examinations are routinely performed at frequent intervals during the nursery course of all preterm infants of 33 weeks or less gestation who are admitted to the intensive care nursery of Thomas Jefferson University Hospital. After discharge, the following survivors during the past 5 years had repeated ultrasound examinations until the anterior fontanel closed and clinical assessments until the presence or absence of cerebral palsy at a minimum age of 12 months was established: all infants with grade III/IV intracranial hemorrhage, periventricular echodensity and periventricular cyst formation, selected infants with either normal ultrasound findings or grade I/II intracranial hemorrhage. Fifteen survivors were found to have cerebral palsy, and all had at least one of the following ultrasound abnormalities: diffuse, bilateral, and multiple periventricular cysts 3 mm or more in diameter that persisted beyond term age, bilateral asymmetrical dilation of the lateral ventricles following grade III intracranial hemorrhage with small periventricular cysts, and ventricular porencephaly following an ipsilateral grade IV intracranial hemorrhage. The periventricular cysts were usually preceded by extensive echodensities of the white matter surrounding the lateral ventricles; these findings were suggestive of periventricular leukomalacia and were the most common abnormal findings on ultrasound in the infants with cerebral palsy. Of 124 infants without cerebral palsy, 121 had no or less severe abnormal findings on ultrasound; the exceptions were three infants with bilateral persistent large periventricular cysts who had normal motor development in late infancy.     

腹腔镜治疗新生儿卵巢囊肿

目的探讨新生儿卵巢囊肿的治疗方法。方法对腹腔镜手术治疗的13例新生儿及小婴儿卵巢囊肿进行了回顾性总结。7例年龄小于1个月,单侧12例,双侧1例,产前B超发现12例;单纯型6例,复杂型7例。结果手术证实单纯型均无并发症发生,其中5例为新生儿。复杂型中6例并发扭转,其中1例病理诊断双侧卵巢颗粒细胞瘤;另1例复杂型实为卵巢囊性畸胎瘤。所有患儿无术中和术后并发症。结论产前B超发现的卵巢囊肿,复杂型及囊肿大于5cm的单纯型应及时腹腔镜手术。     

Progressive stridor: could it be a congenital cystic lung disease?

Bronchogenic cyst of the mediastinum, a cause of stridor early in life, is the result of abnormal budding of the ventral segment of the primitive foregut. Bronchogenic cysts are often asymptomatic in older children and adults. However, symptomatic cases usually manifest early in life with cough, stridor or wheezing due to airway compression. We report a female infant aged 4.5 months with a normal full-term pregnancy, who developed respiratory distress with stridor. This stridor was preceded by a history of slowly progressive noisy breathing. Physical examination revealed evidence of bilateral obstructive emphysema. Chest radiograph revealed bilateral overinflation. Fibro-optic bronchoscopy revealed posterior mediastinal compression. Possibility of congenital cystic lung disease (CCLD) was considered, emphasizing the value of computed tomography (CT) chest, which revealed a cyst probably bronchogenic. Surgical excision was performed with evident histological confirmation of bronchogenic cyst.
Conclusion:  we highlight that in any infant, presented with slowly progressive noisy breathing in the first year of life, CCLD should be considered in the differential diagnosis even with normal X-ray chest. CT chest should be performed for exclusion or diagnosis of the case.